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The nature and prevalence of catatonic symptoms in young

people with autism

Breen J. & Hare D.J. (2017)

In press Journal of Intellectual Disability Research

DOI 10.1111/jir.12362

Abstract

Background A proportion of young people with autism are reported to show catatonic-

like symptoms in adolescence. The aetiology and prevalence of such presentations is

unknown but include a set of behaviours that can best be described as attenuated.

Method The current study empirically investigated the presence and nature of such

attenuated behaviours in children and adolescents with autism using a newly developed

34-item third-party report measure, the Attenuated Behaviour Questionnaire. Caregivers

or parents of young people with autism reported on the presentation of symptoms via

the online completion of the Attenuated Behaviour Questionnaire and two established

clinical measures of repetitive behaviour and depression.

Results Initial results indicate that the Attenuated Behaviour Questionnaire is a

workable clinical measure in this population with a degree of discriminant validity with

regard to catatonia. Attenuated behaviour indicative of catatonia were relatively

common in young people with autism with up to 20.2% having an existing diagnosis of

catatonia and evidence of a relationship between attenuated behaviours and measures of

depression and repetitive and restricted behaviours.

Conclusion Catatonic symptoms are more prevalent in young people with autism than

previously thought and the Attenuated Behaviour Questionnaire has potential as a

clinical and research tool.

Keywords: autism; catatonia; attenuated behaviour

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Introduction

Catatonia is characterised by abnormal hyper- or hypo-kinetic movements and

behavioural alterations (Fink 1994; Bräunig, Krűger, Shuger, Höffler & Börner 2000)

and is diagnosed on the presentation of positive and negative symptoms including

mutism, catalepsy, facial grimaces, catalepsy, echolalia and akinesia in both

International Classification of Diseases 10th Edition [ICD-10] (World Health

Organization 1992) and Diagnostic and Statistical Manual of Mental Health Disorders

5th Edition [DSM-V] (American Psychiatric Association 2014). In severe cases, there

is a sudden and dramatic loss of functional skills with subsequent difficulties with

personal care, expressive communication and engagement in activities. Catatonia is

rarely diagnosed in isolation and is usually co-morbid with other disorders including

depression (Starkstein et al. 1996), encephalitis (Shill & Stacy 2000) and in

developmental disorders including autism spectrum disorder [ASD] (Wing & Shah

2000) and Prader-Willi syndrome (Dhossche & Bouman 1997). Despite this, catatonia

continues to be associated with psychosis by clinicians, despite being more prevalent in

other clinical populations (Fink et al. 2010) and most people with psychosis are not

catatonic (Heckers, Tandon & Bustillo, 2010) and there is a call for catatonia to be

regarded as an independent syndrome or spectrum (Rosebush & Mazurek 2010). In

order to accurately assess catatonia, several catatonia rating scales have been developed

including the Bush-Francis Catatonia Rating Scale (Bush, Fink Petrides, Dowling

& Francis 1996), the Northoff Catatonia Scale (Northoff, Wenke, Demisch Eckert,

Gille & Pflug 1995) and the Bräunig Catatonia Rating Scale (Bräunig et al. 2000).

Although these instruments have good inter-rater and test-retest reliability (Sienhart et

al. 2011), the lack of a ‘gold standard’ definition of catatonia has restricted the

development of catatonia rating scales and subsequent research (Dhossche & Rout.

2006; Sienhart, Rooseleer & De Fruyt 2011). Moreover,

Carroll, Kirkhart, Ahuja, Soovere,, Lauterbach, Dhossche et al. (2008) propose that

individual rating scales are required for the different populations of catatonic patients

and there are problems with the rating scales, including inconsistent item definitions, a

range of items (21-54), differing thresholds for diagnosis, omission of the affective

symptoms (cf Abrams & Taylor 1976) and limited measurement of severity and

frequency of symptoms. Moreover, none of these measures are validated for use with

people with intellectual and developmental disorders and there are practical issues

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including over-reliance on affective alterations that are difficult to accurately identify

via third-party report and on alterations in speech or tone of voice and movement that be

a feature of the developmental disorder itself (Carroll et al. 2008; Heckers, Tandon &

Bustillo 2010)

In the case of ASD, there are reports of markedly abnormal motor movement, including

extreme slowness in executing purposive movement, increased passivity, freezing

during motor movement and difficulty initiating actions (Wing & Shah 2000; Hare &

Malone 2004; Wing 2005) and such ‘autistic catatonia’ is usually onset in adolescence

with a detrimental effect on quality of life (Wing & Shah 2006). Clinical accounts of

autistic catatonia report hindered movements either in fluidity or quantity, remaining

immobile for long periods of time, finding it difficult to start moving and performing

movements very slowly or getting ‘stuck’ part way through a gross motor action (Wing

& Shah 2000; Hare & Malone 2004), with physical and verbal prompts often being

required (Hare & Malone 2004). Such apparent catatonia varies in presentation (Wing

& Shah 2000; Billstead et al. 2005) and severity (Dhossche et al 2006) and is usually

chronic (Ohta et al. 2006; Dhossche et al. 2006) but with some reports of cyclic

(Realmuto & August 1991) and diurnal (Ohta et al. 2006) presentations. However, there

is no commonly accepted diagnostic definition for ‘autistic catatonia’ and across the

various case descriptions (Realmuto & August 1991; Dhossche 1998; Hare & Malone

2004; Ghaziuddin et al. 2005; Ohta et al. 2006; Schieveld 2006; Takota & Takata 2007;

Kakooza-Mwesigeet al. 2008), the number and severity of presenting symptoms varies

and few display all of the possible commonly associated symptoms (Wing & Shah

2000). Some symptoms are commonly reported (e.g. reduced communication, slow

motor movements, resistance to prompting, reduced eating) and others reported only

once (e.g. visual hallucinations, diaphoresis, spontaneous crying) [Appendix 1].

In their study of the prevalence of catatonic symptoms in 506 referrals to a specialist

ASD clinic, Wing and Shah (2000) proposed a set of diagnostic guidelines for autistic

catatonia, including four ‘essential features’ - increased slowness affecting movement

and verbal responses, difficulty in initiating and completing actions, increased reliance

on physical or verbal prompts and increased passivity and apparent lack of motivation

along with four frequently observed behavioural abnormalities - reversal of day and

night, Parkinsonian features, ‘excitement and agitation’ and increase in repetitive,

ritualistic behaviour. Subsequent researchers have adapted these criteria by adding and

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removing specific diagnostic items, whilst the DSM-IV definition of catatonia (APA,

1994) has been used by a number of researchers (Dhossche & Bouman 1997;

Ghaziuddin et al. 2005; Schieveld 2006; Bozkurt & Mukaddes 2010). Other researchers

have proposed their own diagnostic criteria for autistic catatonia (Hare & Malone 2004;

Ohta et al. 2006; Fink et al. 2006). A systematic investigation of such presentations is

clearly required for research and clinical purposes, especially as prevalence is estimated

at 6-17% (Wing & Shah 2006; Billstedt et al. 2005; Perisse et al. 2010; Nordin &

Gillberg 1998) and catatonia is now specifically referenced in DSM-V (American

Psychiatric Association 2013). Treatment options are limited (Ohta et al. 2006) and a

recent systematic review of the treatment of ‘autistic catatonia’ (DeJong et al. 2014)

indicated a paucity of evidence for all of the treatment modalities (behavioural,

pharmacological and electro-convulsive therapy) reviewed.

Given the heterogeneous presentation and lack of consensus regarding aetiology and

treatment with regard to ‘autistic catatonia’, it is proposed that the term attenuated

behaviour is both accurate and more useful to researchers and clinicians and the current

paper reports on the development of a novel measure of attenuated behaviour in young

people with ASD.

Methods

Measures

The following measures were used in the study:

Attenuated Behaviour Questionnaire (ABQ) – a 34 item, third-party report measure

was specifically designed for the present study to determine the prevalence and

frequency of attenuated behaviours typically associated with ‘autistic catatonia’. A

literature search was completed using the on-line databases ‘Psychinfo’ and ‘Web of

Knowledge’, which were systematically searched using key words autism, Autistic

Spectrum Disorder, ASD, catatonia, the wildcard autis* and the exact phrase “autistic

catatonia”. To ensure the literature search was not subjected to a publishing bias,

efforts were made to locate relevant undergraduate theses and poster presentations

together with appropriate hand searching of journals. The numbers of each reported

symptoms connected to autistic catatonia in the research literature were tallied

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(Appendix 1). Any symptom reported only once (e.g. finger tapping, diaphoresis) was

excluded from the measure as was any symptom which is a key feature of ASD and/or

which did not indicate change to the individual’s previous presentation were also

excluded (e.g. echolalia). Any symptoms that were not amenable to accurate and

reliable third-party report (e.g. auditory hallucinations, anxiety, visual hallucinations)

were also excluded as any item that formed part of the other measures administered in

the present study. A total of 88 reported symptoms were thus excluded.

Existing measures of catatonia were studied and their structure replicated as

appropriate. Using this approach, the ABQ was developed consisting of 34 items,

categorised as motor symptoms (n=15), affective alterations (n=5) and behavioural

alterations (n=14), in a similar way to the Northoff Catatonia Scale (Northoff et al.

1995). Each symptom was defined with examples and behavioural descriptions to

address concerns about inconsistent or vague symptom definitions in extant catatonia

rating scales (Carrol et al. 2008). The items included in the ABQ, the order of

presentation in the measure (‘ABQ question number’) the category assigned by the

author (‘motor symptoms’, ‘affective alterations’ or ‘behavioural alterations’) and the

associated examples and descriptors of symptoms provided to participants are shown in

Appendix 2.

Items on the ABQ are rated on a five point scale designed to capture the presentation of

a symptom over time, with symptoms that are progressively worsening over time being

scored more highly:

0=-No, never

1= No, not at the moment but it used to happen

2=Yes but less than before

3=Yes, the same as before

4= Yes, more than before

The six most commonly reported autistic catatonia symptoms, representing difficulty

with motor movement (ABQ item numbers 1-6 in Appendix 2) were termed ‘core

symptoms of autistic catatonia’ as they are considered to be essential for diagnosis.

Positive responses to these items (i.e. being rated 2, 3 or 4) triggered two

supplementary questions measuring current frequency (i.e. the usual amount of time the

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symptom is present during waking hours) and severity (i.e. the effect of the symptom on

the individual’s ability to perform tasks or activities) of the symptom:

. How often does the individual experience these periods of [symptom] at the moment?

Choose from the following options:

• All or almost all of the time they are awake

• Most of the time they are awake

• Some of the time they are awake

• Rarely when they are awake

1) How severely does the individual experience these periods of [symptom] at

the moment?

Choose from the following options:

• Very severely – they seem unable to focus on or do anything else at these

times

• Quite severely – it is difficult for them to focus on or do anything else at

these times

• Moderately – there seems to be an effect on their ability to focus on or do

things

• Slightly – this seems to have little or no effect on their life

The pilot ABQ was examined by non-native English speakers prior to recruitment to

ensure clarity and simplicity of the language used.

Repetitive Behaviour Questionnaire [RBQ] (Moss, Oliver, Arron, Burbidge & Berg.

2009) - a 19 item third party measure assessing prevalence and phenomenology of

restricted and repetitive behaviours in people with intellectual disabilities with item is

rated as occurring ‘more than once a day’, ‘once a day’, ‘once a week’, ‘once a month’

or ‘never’. The items are scored into five sub-domains; ‘Stereotyped behaviour’,

‘Compulsive behaviour’, ‘Restricted preferences’, ‘Repetitive speech’ and ‘Insistence

on sameness’ and a total score can be obtained from the sum of the sub-domains. The

RBQ has good concurrent and construct validity when used with people with ASD aged

3-16.5 years (Honey et al. 2012). The RBQ was included in the study given the

potential for phenomenological overlap between the various forms of repetitive and

restricted behaviour as measured by the RBQ and features of autistic catatonia.

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Carer Supplement to the Glasgow Depression Scale for people with Learning

Disability [GDS-CS] (Cuthill, Espie & Cooper 2003) – a 16 item third-party measure

which assessing current presentation of co-morbid depression in people with intellectual

disabilities. Each item is rated as ‘never/no’, ‘sometimes/a little’ or ‘always/a lot’. The

items are scored in 0-2 format and a cut-off point of 13 for clinical depression. The

GDS-CS correlates with established measures of depression (r=0.88) and has high

content validity, discriminant validity and criterion validity (Cuthill et al. 2003). The

GDS-CS was included given the potential for phenomenological overlap between

autistic catatonia and the psychomotor retardation component of depression, particularly

as the latter is likely to particularly evident to parents and carers.

In addition to descriptive statistics relating to ABQ-derived scores in the present

sample, further analyses were performed to determine:

The relationship of ABQ-derived scores to the reported presence/absence of

extant diagnoses of autistic catatonia

A clinical cut-off for identifying cases of autistic catatonia via a ROC analysis

of a derived total ABQ score.

The association of ABQ-derived scores with demographic variables and with

GDS-CE and RBQ scores

Participants

Informants were recruited via specialist care providers, parent support groups and

charities and were included in the study if they were parent or long-term carer (>2

years) of a young person aged 12-25 years with an existing diagnosis of ASD. A

minimum of 80 participants was required to test the internal validity of the ABQ.

Participants completed measures anonymously via an online questionnaire (Select

Survey) by URL link to a secure website hosted by the University of Manchester. Mean

completion time was 19.7 minutes (range 5-79 minutes). No participants requested help

completing the ABQ, which was taken as indicating that it was clear, readable and easy

to use. The study was reviewed and approved by the University of Manchester

Committee on the Ethics of Research on Human Beings.

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Results

Data were analysed with Statistical Package for the Social Sciences (SPSS) version

20.0. Prior to conducting the analyses, the data was examined to ensure that all

participants met inclusion criteria and that parametric data analysis was permissible.

The majority of participants the informants were parents (91.9%). There were no

missing data but 12 informants dropped out of the study after completing at least up to

item six on the ABQ. A total of N=87 informants completed the full questionnaire and

an additional N=12 informants completed the demographics information and the six

core ABQ.

Table 1 here

41 respondents (41.4%) reported additional diagnostic labels in addition to ASD

resulting in a total of 77 different co-morbid diagnoses (excluding catatonia). N=20

reported an existing diagnosis of catatonia, of whom N=9 had additional diagnostic

labels. Further analysis was not feasible due to the small sample size, but there were

significantly more females than males with diagnoses of catatonia (t (99) =-1.53,

p=0.006). N=84 (85%) reported at least one core symptom either currently or in the past

(Figure 1) with a bimodal distribution of 0 and 3 symptoms.

Figure 1 here

Independent samples t-tests indicated that subjects with extant diagnoses of catatonia

displayed significantly more core symptoms (mean=3.10, SD=1.86) compared to those

without a diagnosis (mean=2.20, SD=1.81) (t (97) =1.97, p=0.05; d=0.49). All six core

symptoms were commonly reported, with difficulty initiating movement (ABQ item 2)

being least reported (n=18) and physical and/or verbal prompts required (ABQ item 6)

most reported (n=60).

Further preliminary analysis of the core symptoms involved computing four summary

variables (ABQ-CAB, ABQ-CS, ABQ-CF & ABQ-TOT) as described in Table 2:

Table 2 here

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The ABQ-CAB scores were bi-modally distributed (0 and 9) with a mean of 7.48

(SD=5.70; range 0-24) and N=15 subjects had an ABQ-CAB score = 0 (15.2%) i.e. no

reported core symptoms to date. There was no significant difference in ABQ-CAB

scores between those with (mean=9.35, SD=6.00) and without (mean=7.01, SD=5.57)

an existing diagnosis of autistic catatonia [t(97)=1.65, p=0.10] whereas those currently

displaying three or more core symptoms had a significantly higher ABQ-CAB scores

(mean=12.04; SD=4.24) than those displaying less than three (mean=3.2; SD=2.86)(

t(97)=-12.23, p<0.01; d=2.44). The Mean ABQ-CS score was 5.23 (SD=4.81; range of

0-23). No ABQ-CS score was recorded for N=22 participants. There was no significant

difference in mean ABQ-CS score between those with (mean=6.95, SD=4.70) and those

without (mean=4.80, SD=4.77) an existing diagnosis of catatonia [t (97) =1.81, p=0.07].

Subjects who currently displayed three or more core symptoms had significantly higher

ABQ-CS scores (mean=8.94; SD=4.09) than those displaying less than three

(mean=1.75; SD=2.01) (t (97) =-11.20, p<0.01; d= 2.23).

The mean ABQ-CF score was 4.73 (SD=3.87; range 0-15) with no ABQ-CF score

being recorded for N=21 participants. Subjects with an existing diagnosis of catatonia

had a significantly higher mean ABQ-CF score (mean=6.45, SD=3.90) than those

without (mean=4.34, SD=3.72) (t(97)=2.24, p<0.05; d=0.55) and those displaying three

or more core symptoms had a significantly higher mean ABQ-CF score (mean=7.85;

SD=2.78) than those displaying less than three (mean=1.86; SD=1.93) (t(97)=-12.51,

p<0.01; d= 2.50).

The mean ABQ-TOT score was 54.28 (SD=21.23; range 5-110) with no significant

differences in mean ABQ-TOT scores between those with (mean=59.33, SD=19.77)

and without (mean=52.96, SD=21.54) existing diagnoses of catatonia [t(85)=1.14,

p=0.259], but those currently displaying three or more core symptoms had a

significantly higher ABQ-TOT score (mean=61.04; SD=21.07) than those displaying

less than three (mean=44.33; SD=22.02) ( t(82)=-3.53, p<0.01; d= 0.77).

A clinical cut-off score for identifying cases of autistic catatonia was derived from a

Receiver Operating Curve (ROC) analysis using the ABQ-CAB scores (Appendix 3).

The area under curve was computed as 62.4% and participants with a diagnosis of

catatonia group was identified by the ABQ-CAB score at significantly higher than

chance (p<0.05), Analysis of the co-ordinates of the ROC curve (sensitivity and

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specificity values) indicated that a conservative clinical cut-off point for catatonia was

an ABQ-CAB score greater than 8.

All ABQ items can be assigned to one of three sub-domains, motor symptoms (n=15),

affective alterations (n=5) or behavioural alterations (n=14). Sub-domain total scores

were derived from the scores (0-4) for the items in each sub-domain. In the current

sample (N=87), mean ABQ Motor Score [ABQMS] was 20.61 (SD=11.25; range 0-55),

mean ABQ Affective Alterations Score [ABQAAS] was 11.30 (SD=4.42; range 0-20)

and mean ABQ Behavioural Alterations Score [ABQBAS] was 22.37 (SD=8.78; range

3-47) and all were positively correlated with ABQ-CABS.

As autistic catatonia appears to onset in adolescence, the older individuals in the sample

are more likely to have catatonia but independent samples t-tests revealed no significant

different in age between those with (mean=16.20, SD=4.420) and those without

(mean=15.55, SD=3.89) an existing diagnosis of catatonia [t (96) = -0.647, p=0.519]

and there were no significant differences between those currently displayed three or

more core behaviours (mean=16.32, SD=4.57) and those who displayed less than three

core behaviours (mean=15.10; SD=3.29) for age; t (96) =-1.53, p=0.130. Pearson Chi-

Square analysis of catatonia diagnoses and gender indicated that these were independent

of each other [Χ² (1, n=99) =0.45, p=0.501] and independent samples t-tests indicated

no significant difference in ABQ-CAB between males (mean=8.89, SD=5.15) and

females (mean=8.64, SD=5.26) in the sample [t (82) =0.195, p=0.846]. Similarly, there

no gender differences were found for the ABQ-CS or ABQ-CF scores.

The mean GDS-CS score for the study sample was 10.56 (SD=5.98) and the scores

range from 0-20. Between- group analyses showed no statistically significant difference

in mean GDS-CS score between those with (mean=13.33, SD=5.02) and those without

(mean=12.77, SD=4.17) an existing diagnosis of autistic catatonia for GDS-CS scores;

t(85)=-0.491, p=0.625) but subjects currently displaying three or more core symptoms

had a significantly higher GDS-CS score (mean=12.74; SD=5.07) than those who

displayed less than three core symptoms (mean=8.43; SD=6.09); t(85)=-3.586, p=0.01;

d= 0.77). Regression analysis of the correlation between these variables indicates a

significant linear association with ABQ-CAB accounting for approximately 15% of the

variation in GDS-CS Score (r2=0.15, p<0.001).

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The mean RBQ score for the sample was 32.82 (SD=16.73; range1-68) and

independent samples t-test indicates that subjects with an existing diagnosis of autistic

catatonia have had significantly higher RBQ total scores (mean=40.56, SD=15.73) than

those without an existing diagnosis (mean=30.80, SD=16.49) (t(85)=2.256, p<0.05; d=

0.60) and those currently presenting with three or more core symptoms also had

significantly higher RBQ total scores (mean=39.58, SD=13.74) than those presenting

with less than three core symptoms (mean=26.20, SD=16.86) (t(85)=-4.05, p<0.01;

d=0.87) Regression analysis of the correlation between these variables indicates a

significant linear association between ABQ-CABS and RBQ Total Score with ABQ-

CABS, accounting for approximately 12% of the variation in RBQ Total Score (r2=0.12,

p<0.001).

The between-group analyses were repeated for the five RBQ sub-domain scores

(Stereotyped behaviour, Compulsive behaviour, Restricted preferences, Insistence on

sameness and Repetitive speech), which identified that subjects currently presenting

with three or more core symptoms had higher Compulsive behaviour sub-domain scores

(mean=11.37, SD=6.28), Restricted preferences sub-domain scores (mean=7.23,

SD=3.64), Insistence on sameness sub-domain scores (mean=5.95, SD=2.39) and

Repetitive speech sub-domain scores (mean=6.79, SD=3.94). Similarly, subjects with an

existing diagnosis of autistic catatonia have significantly higher Repetitive speech sub-

domain scores (mean=7.39, SD=4.18) than those without (mean=5.12, SD=4.14) (t (85)

=2.07, p<0.05; d=0.54). There were significant positive correlation between ABQ-

CACS and sub-domain scores for Compulsive behaviour (r=0.342, n=87, p=0.001);

Restricted preferences (r=0.358, n=87, p=0.001), Insistence on sameness sub-domain

scores (r=0.323, n=87, p=0.002) and Repetitive speech (r=0.336, n=87, p=0.001).

Overall, it was found that in the present sample, higher numbers of the core symptoms

and severity but not frequency thereof being reported when participants had extant

diagnoses of autistic catatonia. On the basis of a visual inspection of the distribution of

the distribution of the number of reported core symptoms, the presence of any three core

symptoms was taken as a putative clinical cut-off. An alternative clinical cut-off of a

score of 8 on the ABQ-CAB sub-total was also derived using a ROC analysis. In

addition, there was an association between the presence of more than three core

symptoms and higher scores on the GADS-CS and RBQ, with these two scales

accounting for 12 % and 15% of the variance in the ABQ-CAB scores.

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Discussion

The ABQ appears to have potential as a valid and practical clinical measure with a

degree of discriminant validity with the six core attenuated behaviours (ABQ items 1-6)

being used as a clinical screening tool for catatonia in ASD with a proposed diagnostic

cut-off of three or more core attenuated behaviours. It can be noted that these six core

attenuated behaviours are essentially the same as Wing and Shah’s (2000) proposed

diagnostic criteria. In particular, the current results support the notion of catatonia as a

continuum in ASD and that catatonia may be under-identified as N=20 participants had

an existing diagnosis of catatonia whereas N=42 displayed three or more core

attenuated behaviours. There is also evidence of a relationship between attenuated

behaviours and measures of depression and repetitive and restricted behaviours,

although a causal relationship was not determined.

The ABQ permits comparison of the change in presentation of attenuated behaviour

over time due to the scoring metric for each question asking participants to rate each

presenting behaviour compared to its past presentation. Assuming the face and

ecological validity of the ABQ to be reasonable on the basis that the items relate to

directly observable behaviours, the ABQ facilitates analysis of changes in the

presentation of symptoms over time and could therefore would provide information

about the course of catatonia and the effectiveness of any intervention. However, the

ABQ would not indicate when a specific behaviour changes, only that it is more, less or

the same as before.

The data from this initial study indicate that attenuated behaviours associated with

catatonia are common in young people with ASD in line with predictions from the

putative models of catatonia in ASD (Wing & Shah 2006; Fink et al. 2006; Takota &

Takata 2007; Gowen & Hamilton 2013). The data also supports the notion that

previously reported variation in the presentation of catatonia in people with ASD (Wing

& Shah 2000; Billstead et al. 2005; Dhossche et al. 2006; Neumarker 2006) might in

part be due to the absence of empirically derived criteria. Moreover, the number of core

behaviours present in individuals with an existing diagnosis of catatonia varied with

between one and six (mean=3.1), supporting Wing and Shah’s (2000) clinical

observations of heterogeneity in presentation. In the present study, only two individuals

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with an existing diagnosis of catatonia displayed all six core behaviours. However, the

number of core behaviours currently displayed appears to be a good indicator of

catatonia as individuals with an existing diagnosis currently presented with significantly

more core behaviours. However, four individuals without an existing diagnosis of

catatonia presented with all six core behaviours.

The estimated prevalence of catatonia in the current study varied depending on the cut-

off applied. Twenty (20.2%) subjects had an existing diagnosis of catatonia whereas 42

(48.3%) displayed three or more core behaviours (i.e. above the proposed clinical cut-

off point). Therefore, the current data indicate that possible prevalence of catatonia in

the current study sample may exceed previous estimates of 6-14% and support

suggestions of an under-diagnosis of catatonia in young people with ASD. It should be

noted that catatonia was not specified in the information used for recruitment and the

research title indicated that the study was investigating movement problems in young

people with ASD. Additionally, the majority of the sample did not have an existing

diagnosis of catatonia (79.8%).

Statistically significant relationships were found between scores on the ABQ and

measures of depression and repetitive and restricted behaviour (appropriate for the study

population). In the case of the former, these conditions may be co-occurring or this may

be due to the apparent overlap in the items on each measure, specifically reduced eating

(ABQ item 27, GDS-CS item 11), reduced communication or muteness (ABQ item 24,

GDS-CS item 5), increased aggression (ABQ item 18, GDS-CS item 2), avoidance of

contact with others (ABQ item 16, GDS-CS item 3), decreased personal hygiene and/or

concern about appearance (ABQ item 31, GDS-CS item 4), crying (ABQ item 17, GDS-

CS item 6), reduced engagement in preferred activities (ABQ item 21, GDS-CS item 8),

requiring more encouragement (ABQ item 22, GDS-CS item 10) and sleep problems

(ABQ item 26, GDS-CS item 12). As both measures are third-party ratings of

observable behaviours, it may be that a young person with ASD is presenting with

either catatonia or depression, but this is being attributed to either or both conditions as

extant measures lack the sensitivity to determine aetiology. For example, reduced eating

could be due to inability to execute motor action in the body or reduced appetite as a

result of depression, but both causes would present identically and be scored on third-

party measures of catatonia and depression. Further investigation is therefore required

to elucidate the apparent relationship between ABQ and GDS-CS scores. The

14

association between repetitive and restricted behaviours and attenuated behaviour as

measured by the ABQ appears to be more robust and not necessarily artefactual. There

is some duplication of items across both measures, specifically body stereotypy (ABQ

item 8, RBQ item 2) and hand stereotypy (ABQ item 13, RBQ item 3). Overall, the

effect sizes for the significant results were found to be moderate to high.

The current study has a several limitations with respect to both design and execution. It

is possible that relevant material was omitted from the initial literature review, which

was based on key words in the title or abstract of published articles. The research

design relied on retrospective reporting of current symptoms with previous presentation,

which may have compromised the accuracy of the data on frequency and severity.

However, the main focus of the study was on the current presentation of observable

attenuated behaviours and this may have off-set any such bias. The online nature of the

study survey resulted in no direct contact between researchers and participants, which

whilst guaranteeing anonymity for participants also precluded checking against

inclusion criteria and therefore the insertion of pre-participation questions that included

confirmation about inclusion criteria was intended to minimise the possibility of error

prior to completion of the online questionnaire. With regard to the determination of

clinical cut-offs, the composite nature of the ABQ meant that both categorical and

nominal cut-offs were necessary, the latter being computed via the ROC curve analysis,

with the majority of the reported analyses being undertaken using the former cut-off,

which should be regarded at this stage as provisional. Further work is required to

identify which cut-off score is the more valid. Finally, the test-retest and inter-rater

reliability of the ABQ were not assessed as part of the present study and should be

incorporated in future research.

Future research could examine the association between ABQ scores and the degree of

global intellectual disability, expressive language impairment and severity of ASD , all

of which have been identified as potentially risk markers for catatonia development

(Wing & Shah, 2000) as well as investigation of speed of onset of attenuated behaviour

symptoms and precipitating factors such as stress, which have been a priori identified

as of prognostic importance (Shah & Wing 2000; Ghaziuddin et al. 2005; Shah & Wing

2006).

15

The ABQ has the capability to assess the course of catatonia and to empirically measure

the effectiveness of treatment interventions over time to support the development of

evidence-based treatments (DeJong et al 2014) and practice guidelines. It could also

have value as a routine screening tool for early detection of catatonia in adolescents

with ASD

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20

Appendix 1 –Symptoms of autistic catatonia

Symptom Frequency in case

reports

Included in ABQ

‘Freezing’/very still like a statue 6 Yes

Difficulty initiating

actions/’stuckness’/akinesia

6 Yes

Problems stopping actions once

started

3 Yes

Difficulty initiating movement 5 Yes

Slowness in movement 5 Yes

Requires prompts to complete actions 4 Yes

Waxy flexibility 3 Yes

Repetitive body movements 4 Yes

Stiff posturing 9 Yes

Noticeable resting tremor 2 Yes

Increased motor tics 3 Yes

Waving or shaking extremities 2 Yes

Twisting or flicking hands in front of

eyes

2 Yes

Moving in a jerky way 2 Yes

Impulsive/excitable phases 4 Yes

Withdrawal from physical contact 2 Yes

Spontaneous crying, laughing or

screaming

4 Yes

Episodes of aggression 6 Yes

Difficulty passing through doorways 2 Yes

Difficulty crossing lines on the floor 2 Yes

Reduced enjoyment in preferred

activities

4 Yes

Requiring more encouragement to

engage

4 Yes

Unusual gait/posture 5 Yes

Reduced communication/muteness 10 Yes

21

Incontinence 4 Yes

Sleep problems 4 Yes

Reduced eating 6 Yes

Eye rolling/ unusual eye movements 5 Yes

Unusual facial expressions/’grimaces’ 5 Yes

Ignoring instructions 2 Yes

Refusal to bathe or change clothes 2 Yes

Occasional groans or unusual noises 3 Yes

Staring into space/fixed gaze 5 Yes

Unable to lift head 2 Yes

Echolalia 2 No – common feature of

ASD

Finger tapping 1 No – reported once

Diaphoresis 1 No – reported once

Withdrawal 1 No – reported once

Auditory hallucinations 1 No – reported

once/speculative

Auditory hypersensitivity 1 No – reported once/

speculative

Depressed 5 No - too vague or

speculative

Anxious 2 No - too vague or

speculative

Realmuto & August (1991), Dhossche (1998), Zaw et al (1999), Hare & Malone (2004),

Ghaziuddin et al (2005), Wing (2005), Dhossche et al (2006), Dhossche & Rout (2006),

Fink et al (2006), Ohta et al (2006), Schieveld (2006), Shah & Wing (2006), Takota &

Takata (2007), Wing & Shah (2006), Dhossche et al. (2009), Bozkurt & Mukaddes

(2010), Kakooza-Mwesige et al. (2008), Ghaziuddin, Dhossche & Marcotte (2012),

Watchtel et al. (2010)

22

Appendix 2: Attenuated Behaviour Questionnaire

ABQ

question

number:

Symptom: Associated

question in ABQ:

Example or descriptor

given:

Supplementar

y questions re

frequency &

severity

Category: Motor Symptoms

1 ‘Freezing’/very

still like a statue

Are there times when

s/he is very still for

long periods of time,

almost like a statue?

Yes

2 Difficulty

initiating

actions/’stuckne

ss’/akinesia

Does s/he seem to get

‘stuck’ when trying

to do something?

Stopping mid-air half

way through reaching

for something & looking

like they are trying to

move but cannot OR

beginning to pick up a

cup to drink but lifting it

only half way and then

putting it down again

Yes

3 Problems

stopping actions

once started

Does s/he seem to

find it difficult to

stop doing actions

once they have

started them?

Repeatedly putting a

coat on & taking it off

again & again for a long

period of time

Yes

4 Difficulty

initiating

movement

Does s/he seem to

find it difficult

to start moving?

Lying still and looking

like S/he wants to get up

or reach for something

but cannot

Yes

5 Slowness in Does s/he move very Moving very slowly Yes

23

movement slowly and takes a

long time to finish

actions?

when doing things like

picking up a cup to

drink or eating dinner

6 Requires

prompts to

complete

actions

Are there times when

s/he

needs physical and/or

verbal prompts to

complete actions?

Needing someone to tell

them or touch their arm

to enable them to lift a

cup to their mouth to

drink

Yes

7 Waxy

flexibility

Are there times when

if you moved part of

their body, they let

you without taking

much notice of what

you are doing & then

stay in that position

afterwards?

Offer no resistance to

you curling their fingers

into a fist & then keep

their hand curled up

when you moved away

No

8 Repetitive body

movements

Does s/he like to

move their body in

repetitive ways?

Any frequent body

movement such as body

rocking, twisting wrists,

flicking fingers etc

No

9 Stiff posturing Does s/he strike and

hold stiff poses?

No

10 Noticeable

resting tremor

When s/he is

completely relaxed,

does any part of their

body tremble

No

11 Increased motor

tics

Does s/he experience

‘tics’ (speech or

movement)?

Suddenly & repetitively

move their body or

saying a word/phrase in

a way they seem unable

to control

No

24

12 Waving or

shaking

extremities

Does s/he move their

hands or feet in an

odd way?

Twisting, waving or

shaking

No

13 Twisting or

flicking hands

in front of eyes

Does s/he twist or

flick their hands in

front of their eyes?

No

14 Moving in a

jerky way

Does s/he move in a

very jerky way?

No

23 Unusual

gait/posture

Does s/he walk

unusually?

No

Category: Affective Alterations

15 Impulsive or

excitable phases

Is s/he impulsive OR

over-excitable?

No

16 Withdrawal

from physical

contact

Are there periods

where s/he withdraws

from contact with

others?

Does not want to be

hugged or touched by

anyone, shutting

themselves in their

room, sitting under a

table alone etc

No

17 Spontaneous

crying, laughing

or screaming

Does s/he scream, cry

or laugh suddenly for

no reason? (If so,

which?)

No

18 Episodes of

aggression

Is s/he aggressive

towards themselves

or others at times?

(If so, which?)

No

21 Reduced

enjoyment in

preferred

activities

Has s/he lost

enjoyment in their

favourite activities?

Currently gets no

enjoyment from

activities they used to

enjoy or refuses to do

No

25

them

Category: Behavioural Alterations

19 Difficulty

passing through

doorways

Does s/he find it

difficult to walk

through doorways?

No

20 Difficulty

crossing lines

on the floor

Does s/he find it

difficult to walk

across lines on the

floor or changes in

flooring?

Such as from a carpet to

a wooden floor

No

22 Requiring more

encouragement

to engage

Is S/he doing less

than they used to?

It is now harder than it

used to be to encourage

them to do activities

No

24 Reduced

communication/

muteness

Are there periods

where s/he

communicates with

others less or not at

all?

This includes all

communication methods

including reduced

speech or in other

communication such as

PECS etc

No

25 Incontinence Are there periods

where s/he is

incontinent or refuses

to use the toilet when

they used to?

Not using skills that

they have used in the

past and are soiling

themselves when they

would have previously

used the toilet.

No

26 Sleep problems Does s/he have sleep

problems?

Finding it difficult to

get to sleep at night,

wants to sleep in the day

but not at night, getting

little sleep etc

No

27 Reduced eating Are there periods No

26

where s/he refuses to

eat or eats less than

they used to?

28 Eye rolling/

unusual eye

movements

Does s/he move or

roll their eyes

unusually?

Repeatedly rolling their

eyes or repeatedly

looking from left to

right

No

29 Unusual facial

expressions/’gri

maces’

Does s/ he pull

unusual facial

expressions or

grimaces?

No

30 Ignoring

instructions

Does s/he ignore

instructions?

This refers to

instructions that they

understands

No

31 Refusal to bathe

or change

clothes

Does s/he refuse to

wash or change their

clothes?

No

32 Occasional

groans or

unusual noises

Does S/he regularly

make groaning or

other unusual noises?

No

33 Staring into

space/fixed

gaze

Does s/he stare into

space or fix their

gaze onto certain

things?

No

34 Unable to lift

head

Does s/he seem

unable to lift their

head?

Their head looks like it

is too heavy for them to

lift up

No

27

Appendix 3: ROC curve analysis